Relapsing polychondritis in childhood: A case report

被引:0
作者
Li, Peng [1 ]
Chen, Zhipeng [2 ]
Lv, Huaiqing [2 ]
Lin, Liqiang [2 ]
机构
[1] Shandong Second Med Univ, Sch Clin Med, Weifang, Peoples R China
[2] Linyi Peoples Hosp, Dept Otorhinolaryngol, Linyi 276000, Peoples R China
关键词
pediatric; relapsing polychondritis; respiratory involvement; tracheostomy;
D O I
10.1097/MD.0000000000040106
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Rationale:Relapsing polychondritis (RP) is a rare immune-mediated disease that leads to progressive cartilage destruction, notably affecting the ears, nose, and airways. Timely diagnosis is essential to prevent irreversible airway damage and life-threatening complications. This study presents a case of a 12-year-old child diagnosed with RP, emphasizing the critical need for timely identification of RP in children.Patient concerns:A 12-year-old girl was admitted with worsening hoarseness and cough, leading to respiratory distress and severe bradycardia, requiring transfer to the pediatric intensive care unit. After successful resuscitation and tracheal intubation, imaging showed significant subglottic swelling. She received antibiotics, corticosteroids, and nebulization. Despite initial stabilization, she required a tracheostomy 2 months post-discharge due to recurrent dyspnea. Six months later, she developed joint pain and a saddle nose deformity.Diagnoses:The patient was clinically diagnosed with RP in accordance with the diagnostic standards set forth by Micheet et al.Interventions:During the initial phase of treatment, the patient was administered antibiotics, corticosteroids, and nebulization therapy. Due to severe respiratory distress, an emergency tracheostomy was performed by ear, nose, and throat surgeons. After a definitive diagnosis, the patient was treated with tocilizumab, methotrexate, and corticosteroid therapy. Additionally, supportive measures including calcium supplementation, gastric protection, and immune support were provided.Outcomes:Three years post-diagnosis, the patient's condition is stable.Lessons:Due to RP low prevalence, diagnosis in children is frequently delayed or overlooked. Notably, involvement of the major airways is more common and severe in pediatric patients compared to adults. It is crucial for Otolaryngologists to have a comprehensive understanding of this condition to effectively diagnose and manage complications associated with RP.
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页数:3
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共 13 条
  • [1] Relapsing polychondritis in children: A review
    Alqanatish, Jubran T.
    Alshanwani, Jawaher R.
    [J]. MODERN RHEUMATOLOGY, 2020, 30 (05) : 788 - 798
  • [2] Limited auricular relapsing polychondritis in a child treated successfully with infliximab
    Alqanatish, Jubran Theeb
    Alfarhan, Basma Ali
    Qubaiban, Sara Mohammed
    [J]. BMJ CASE REPORTS, 2019, 12 (05)
  • [3] Pediatric-Onset Relapsing Polychondritis: Case Series and Systematic Review
    Belot, Alexandre
    Duquesne, Agnes
    Job-Deslandre, Chantal
    Costedoat-Chalumeau, Nathalie
    Boudjemaa, Sabaa
    Wechsler, Bertrand
    Cochat, Pierre
    Piette, Jean-Charles
    Cimaz, Rolando
    [J]. JOURNAL OF PEDIATRICS, 2010, 156 (03) : 484 - 489
  • [4] Buscatti IM, 2013, ACTA REUMATOL PORT, V38, P208
  • [5] Sustained Remission with Tocilizumab in Refractory Relapsing Polychondritis with Ocular Involvement: A Case Series
    Farhat, Rebecca
    Clavel, Gael
    Villeneuve, Delphine
    Abdelmassih, Youssef
    Sahyoun, Marwan
    Gabison, Eric
    Sene, Thomas
    Cochereau, Isabelle
    Titah, Cherif
    [J]. OCULAR IMMUNOLOGY AND INFLAMMATION, 2021, 29 (01) : 9 - 13
  • [6] Figaro NJ., 2023, Cureus, V15, pe40085
  • [7] Relapsing polychondritis in childhood: three case reports, comparison with adulthood disease and literature review
    Fonseca, Adriana R.
    de Oliveira, Sheila K. F.
    Rodrigues, Marta C. F.
    Aymore, Ierece L.
    Domingues, Romeu C.
    Sztajnbok, Flavio R.
    [J]. RHEUMATOLOGY INTERNATIONAL, 2013, 33 (07) : 1873 - 1878
  • [8] Gorard Camilla, 2014, BMJ Case Rep, V2014, DOI 10.1136/bcr-2014-205036
  • [9] Tocilizumab in VEXAS relapsing polychondritis: a single-center pilot study in Japan
    Kirino, Yohei
    Takase-Minegishi, Kaoru
    Tsuchida, Naomi
    Hirahara, Lisa
    Kunishita, Yosuke
    Yoshimi, Ryusuke
    Nakajima, Hideaki
    [J]. ANNALS OF THE RHEUMATIC DISEASES, 2021, 80 (11) : 1501 - 1502
  • [10] Relapsing polychondritis in childhood - case report and short review
    Knipp, S
    Bier, H
    Horneff, G
    Specker, C
    Schuster, A
    Schroten, H
    Lenard, HG
    Niehues, T
    [J]. RHEUMATOLOGY INTERNATIONAL, 2000, 19 (06) : 231 - 234